• ‘How you keep going’: Voluntary sector practitioners’ story-lines as emotion work

      Quinn, Kaitlyn; Tomczak, Philippa; Buck, Gillian; University of Toronto; University of Nottingham; University of Chester (Wiley, 2022-01-16)
      The voluntary sector acts as the last line of defense for some of the most marginalized people in societies around the world, yet its capacities are significantly reduced by chronic resource shortages and dynamic political obstacles. Existing research has scarcely examined what it is like for voluntary sector practitioners working amidst these conditions. In this paper, we explore how penal voluntary sector practitioners across England and Scotland marshaled their personal and professional resources to “keep going” amidst significant challenges. Our analysis combines symbolic interactionism with the concept of story- lines. We illuminate the narratives that practitioners mobilized to understand and motivate their efforts amidst the significant barriers, chronic limitations, and difficult emotions brought forth by their work. We position practitioners' story- lines as a form of emotion work that mitigated their experiences of anger, frustration, overwhelm, sadness, and disappointment, enabling them to move forward and continue to support criminalized individuals. Our analysis details three story- lines— resignation, strategy, and refuge—and examines their consequences for practitioners and their capacities to intervene in wicked social problems.
    • Invisible and at-risk: older adults during the COVID-19 pandemic.

      Benbow, Susan M; Bhattacharyya, Sarmishtha; Kingston, Paul; Peisah, Carmelle (2021-12-16)
      During the COVID-19 pandemic the risks to older adults of systemic abuse and neglect have become amplified, alongside increasing abuse and neglect in the community. Novel risks have also evolved involving cybercrime and the use of remote technologies in health and social care related to the pandemic. This commentary brings together lessons to be learned from these developments and initial ideas for actions to mitigate future risks.
    • Welfare conditionality, ethics and social care for older people in the UK: From civic rights to abandonment?

      Carey, Malcolm; University of Chester (Oxford University Press, 2021-12-13)
      Welfare systems are becoming ever more conditional, with access to state support increasingly rationed via a legion of legally-defined and financially-driven restrictions and rules. Civic protection and economic rights for older citizens within Western policy systems are subsequently diminishing and continue to give way to neoliberal discursive practices which prioritise welfare activation, autonomy, participation, asset-based yet precarious self-care, the aversion of health-centred risks and much higher levels of eligibility for support. This article looks at welfare conditionality and its relationship to older people, ethics and governance within social care. By using three examples of welfare conditional reforms from the UK, it is highlighted that strains typically persist between the altruistic components of some ethical frameworks and the everyday experiences of many older people. The relative gatekeeping powers of welfare professionals and expectations placed on family members and carers have also increased, especially upon older people with higher needs and who may lack economic and cultural capital. This is despite rhetorical policy-led claims of increasing choice and control, and allowing support to be more asset-based and personalised.
    • The supervisor conundrum

      Knight, Kate; Leigh, Jacqueline; Whaley, Viki; Matthews, Marie; Doyle, Kate; Rabie, Gay; University of Chester; University of Salford; Manchester University NHS Foundation Trust (MA Healthcare, 2021-11-11)
      Commentry
    • The potential impact of extensive privatisation in the UK upon the ‘life chances’ of young people in care

      Carey, Malcolm; University of Chester (National Youth Agency, 2021-11-01)
      The article considers the potential impact of extensive privatisation in the UK upon the ‘life chances’ of young people in care
    • This is how it feels: activating lived experience in the penal voluntary sector

      Buck, Gillian; Tomczak, Philippa; Quinn, Kaitlyn; University of Chester; University of Nottingham; University of Toronto (Oxford University Press, 2021-10-21)
      Increasing calls for ‘nothing about us without us’ envision marginalised people as valuable and necessary contributors to policies and practices affecting them. In this paper, we examine what this type of inclusion feels like for criminalised people who share their lived experiences in penal voluntary sector organisations. Focus groups conducted in England and Scotland illustrated how this work was experienced as both safe, inclusionary and rewarding and exclusionary, shame-provoking and precarious. We highlight how these tensions of ‘user involvement’ impact criminalised individuals and compound wider inequalities within this sector. The individual, emotional and structural implications of activating lived experience therefore require careful consideration. We consider how the penal voluntary sector might more meaningfully and supportively engage criminalised individuals in service design and delivery. These considerations are significant for broader criminal justice and social service provision seeking to meaningfully involve those with lived experience.
    • Assessment of sodium and iodine intake among university students in Casablanca, Morocco

      Jafri, Ali; Elarbaoui, Maria; Elkardi, Younes; Makhlouki, Houria; Ellahi, Basma; Derouiche, Abdelfettah; Université Mohammed VI des Sciences de la Sante; University of Chester; Université Hassan II de Casablanca (Elsevier, 2021-07-08)
      Introduction. – Iodine deficiency is still a matter of public health concern despite salt fortification andespecially with global recommendations to lower salt intake, this is mainly due to dietary habits. Uni-versity students have a diet based on street food high in sodium and low in other micronutrients (i.e.iodine and potassium). In this study, we aim to measure sodium and iodine levels in university studentsto assess their risk of developing complications later in life.Methodology. – A sample of 120 students aged between 18 and 25 years old was recruited and asked tocollect their 24-hours urine samples in special containers containing. Samples were stored then analyzedfor sodium, potassium, iodine and creatinine levels.Results. – The average urinary excretion of sodium was 3066.8 ± 1196.0 mg/day. Overall, 72.6% of par-ticipants consume more than 2 g/day of sodium. Average potassium intake is 1805.9 ± 559.4 mg/day,and all participants consume less than the adequate amount. Daily urinary excretion of iodine is135.6 ± 88.9 mg/day, and 69.2% of participants consume less than the recommended amount. Sodium,potassium and iodine intakes were higher in male participants (P-values = 0.008; 0.044 and 0.003, respec-tively). The lowest average iodine intake was observed in underweight participants (119.4 ± 31.4) with87.5% of underweight participants and 80% of female participants below the recommended intake.Conclusion. – Sodium intake is high while iodine intake is low in this studied population, especially inwomen.
    • Universal credit, lone mothers and poverty: some ethical challenges for social work with children and families

      Carey, Malcolm; University of Chester (Taylor and Francis, 2021-06-22)
      This article critically evaluates and contests the flagship benefit delivery system Universal Credit for lone mothers by focusing on some of the ethical challenges it poses, as well as some key implications it holds for social work with lone mothers and their children. Universal Credit was first introduced in the United Kingdom (UK) in 2008, and echoes conditionality-based welfare policies adopted by neoliberal governments internationally on the assumption that paid employment offers a route out of poverty for citizens. However, research evidence suggests that the risks of conditionality polices for lone parents can often include increased poverty, a deterioration in mental health or even destitution posed by paternalistic sanctions or precarious low-paid employment, which can undermine parenting capacities and children’s well-being. The article also critically appraises and questions challenges posed by an increased reliance upon contractual ethics by governments, alongside the wider behaviour modifying policies of the workfare-orientated state. This includes that working-class lone mothers can erroneously be stigmatised as representing a morally challenged dependent burden through activation policies and risk-averse social work practices.
    • Decision making in the management of adults with malignant colorectal polyps: An exploration of the experiences of patients and clinicians

      Westwood, Clare; Lee, Tom; McSherry, Robert; Bettany-Saltikov, Josette; Catlow, Jamie; University Hospital North Tees and Hartlepool NHS Foundation Trust; North Tyneside General Hospital; University of Chester; Teesside University (Wiley, 2021-05-18)
      Aim: A diagnosis of colorectal polyp cancer presents a treatment dilemma. The decision between segmental resection versus endoscopic surveillance is difficult due to lack of good quality clinical evidence for either option. The aim of this study was to understand the decision-making experiences of both clinicians and patients when faced with such a diagnosis. Methods: Qualitative, semi-structured interviews were undertaken with ten clinicians involved in the care of patients diagnosed with polyp cancer and five patients who had experience of a diagnosis of polyp cancer. All clinicians and patients were from four hospital Trusts across the North of England. Interviews were audio recorded, transcribed verbatim and analysed using the principles of Interpretative Phenomenological Analysis. Results: Analysis of the interview transcripts evidenced the difficulties faced by both groups when faced with treatment decisions following a diagnosis of colorectal polyp cancer. Some of these difficulties were specific to either the clinician or patient group. Themes which were common to both groups included: complexity of risk information; external influences, unexpected diagnosis; and time. In addition, hospital system factors were disclosed which also influenced clinician and patient experiences. Conclusion: This research study has evidenced several factors such as uncertainty, complexity of risk information and influences on decisions which are preventing patients being fully involved in treatment decisions following a diagnosis of colorectal polyp cancer. Recommendations for improvements in practice, including a framework to assist treatment decision making in the future have been highlighted. What does this paper add to the literature? This qualitative study is, to the authors knowledge, the first exploring clinician and patient experiences of treatment decision making following a colorectal polyp cancer diagnosis. Key factors influencing how treatment decisions are made have been identified. As a result, a framework is proposed highlighting critical factors for consideration to deliver patient centred care.
    • Employing with conviction: The experiences of employers who actively recruit criminalised people

      Atherton, Peter; Buck, Gillian; University of Chester (SAGE Publications, 2021-05-03)
      In England and Wales, criminal reoffending costs £18 billion annually. Securing employment can support desistance from crime, but only 17% of ex-prisoners are employed a year after release. Understanding the motivations of employers who do recruit criminalised people therefore represents an important area of inquiry. This article draws upon qualitative interviews with twelve business leaders in England who proactively employ criminalised people. Findings reveal that inclusive recruitment can be (indirectly) encouraged by planning policies aimed to improve social and environmental well-being and that employers often work creatively to meet employees’ additional needs, resulting in commercial benefits and (re)settlement opportunities.
    • Critical analysis of the Armed Forces Covenant Fund Trust Aged Veterans Fund

      Di Lemma, Lisa C G; orcid: 0000-0001-9161-1779; Finnegan, A; orcid: 0000-0002-2189-4926; Howe, S (BMJ Publishing Group, 2021-03-30)
      Background: Relatively little research is available regarding the specific needs of older military veterans and the services introduced to support them. In 2016, the Armed Forces Covenant Fund Trust launched the Aged Veterans Fund (AVF), to understand the impact that military service may have on ageing, and to support initiatives targeting their health and well-being. This fund was financed for 5 years and included 19 UK portfolio projects. Method: The paper presents a retrospective evaluation on the processes and impact of the AVF, with the intent of informing policy, educational services, service providers and stakeholders of the lessons learnt. The inclusion criteria was veterans and their families aged 65 years of age or over. In 2019, data were drawn from documentary evidence related to the programmes. Qualitative analysis were performed on 78 eligible sources and 10 themes were identified. Results: Programmes were rolled out via collaborative partnerships referrals, focusing on person-centred or skill-exchange approaches. Challenges were encountered, such as capacity and timelines issues. A limited amount of associated cost-savings was observed, even if examples of sustainability and high satisfaction were reported. Evidence was found of programmes boosting health and well-being outcomes, in raising awareness, and in positively impacting on clinical practice, such as re-admission rates. Conclusion: The AVF programmes were successful in their intent to provide support to older veterans and their families. The findings provide indicators of the next steps required for the support of ageing veterans. Further investigation of the cost-effectiveness of age-friendly veterans’ services is needed.
    • The Teaching of Psychological Theory in the Undergraduate Pre-Registration Nurse Training Curriculum: Systematic, Integrative Literature Review

      Mitchell, Andrew E P; University of Chester (Nova Science Publishers, 2021-03-30)
      Aims and objectives. To establish how best to integrate psychology education into the pre-registration nurse training curriculum to enhance clinical practice. Background. Educational psychology focuses on applications of science to understand and improve how students learn and how they are taught. A key challenge for academics is integrating psychological theory within teaching sessions and clinical practice. Didactic teaching methods have had limited success as students do not see the direct relevance of psychological theory for clinical practice. Problem-based learning and simulation sessions may enhance the perceived importance for clinical practice. Design. Systematic, integrative literature review. Methods. A systematic search of the literature using multiple databases and search engines between the years 2010-2020 was undertaken using keywords and PICO algorithm. For this study, the following keywords were utilised; student nurse, pre-registration, education, problem-based learning, practice skills, simulation, psychology and learning theory. PICO identifiers were (Participants) pre-registration students, (Intervention) – psychology and psychological learning theory, (Comparison) – didactic taught sessions with problem-based and simulation, (Outcome) - improvement in theory or practice-based assessment. Results. Eleven studies were included. Evidence for traditional didactic teaching is limited. There is evidence that problem and simulation-based learning has shown success in demonstrating clinical practice implications. Conclusions. The findings reveal that psychology education is considered a central aspect of nurse training. Observational research is required to understand better the link between psychological knowledge and clinical practice. Relevance to clinical practice. There should be a strategic focus on the development and implementation of a coherent psychological theory in the pre-registration nurse training curriculum. Coherent and applied psychology curricula may have clear benefits for nurse education and clinical practice.
    • Evidence-based practice and evidence-informed practice competencies in undergraduate pre-registration nursing curricula: a document analysis at a University in England

      Kumah, E; Bettany-Saltikov, J; van Schaik, P; McSherry, R; Teesside University; University of Chester (Elsevier, 2021-03-13)
      Evidence-based practice and evidence-informed practice competencies in undergraduate pre-registration nursing curricula: a document analysis at a University in England Abstract Background In response to the heightened emphasis on incorporating the best available evidence into healthcare decision-making, healthcare training institutions have been actively incorporating Evidence-Based Practice (EBP), and/or Evidence-Informed Practice (EIP) competencies into undergraduate healthcare curricula. However, there is a gap in the scientific knowledge about the actual contents, as well as the extent of integration of EBP and EIP in undergraduate pre-registration nursing programmes. Method A document analysis utilising Rohwer et al.’s (2014) framework was conducted to review and analyse the content of EBP and EIP competencies in the 2018/2019 curriculum of the undergraduate pre-registration nursing programme of a University located in England, United Kingdom. Results Competencies relevant to EBP were included in four nursing modules. However, EIP competencies were not included in the curriculum. Conclusion There is an urgent need for a more structured and holistic way of teaching and assessing EBP competencies through the integration of the principles of EIP, in order to enhance the effective application of evidence into clinical nursing practice.
    • A narrative review of literature on the use of health and social care by older trans adults: what can United Kingdom services learn?

      Benbow, Susan Mary; Eost-Telling, Charlotte; Kingston, Paul; University of Chester; Older Mind Matters (Cambridge University Press, 2021-02-24)
      We carried out a narrative review and thematic analysis of literature on the physical healthcare, mental healthcare and social care of trans older adults to ascertain what is known about older trans adults’ contacts with and use of health and social care. Thirty papers were found: a majority originated in the United States. Five themes were identified: experience of discrimination/ prejudice and disrespect; health inequalities; socioeconomic inequalities; positive practice; and staff training and education. The first three themes present challenges for providers and service users. Experiences of discrimination/ prejudice and disrespect over the course of their lives powerfully influence how older trans adults engage with care services and practitioners. Health and socio-economic inequalities suggest that older trans adults are likely to have greater need of services and care. The remaining two themes offer opportunities for service improvement. We conclude that more research is needed, that there is a strong argument for taking a life course perspective in a spirit of cultural humility, and that contextual societal factors influence service users and providers. We identify positive trans-inclusive practices which we commend to services. More needs to be done now to make older adult services appropriate and welcoming for trans service users.
    • The use of photo elicitation to explore the impact of social work student’s perceptions of placements on social work tutors and consider their role in practice learning

      Caffrey, Bridget; Fruin, Helen; Bailey-McHale, Julie; Ridgeway, Victoria; Bailey-McHale, Bex; University of Chester (Taylor & Francis, 2020-12-22)
      The importance of learning in practice is acknowledged across health and social care professions. Social work students’ experiences in practice settings has attracted some attention in academic literature, and the role and impact of the Practice Educator (PE) on student learning is increasingly recognised. However, there is a paucity of research examining the role of the social work tutor generally and particularly within practice learning settings. This paper presents a small-scale qualitative study exploring the impact of visual images produced by social work students reflecting their practice experiences on six social work tutors. Photo elicitation prompted discussion in a focus group setting which was subsequently thematically analysed, with four themes emerging. These were dichotomous relationships, difference and diversity, tutor brokerage skills, and student support. The images encouraged tutors to reflect upon the complexity of their relationship with social work students and question whether they were professionally equipped to support students in complex placement situations. In addition, the effectiveness of the curriculum in preparing social work students for practice was considered, particularly space afforded to students to reflect on practice learning in a safe environment. Opportunities to support SW tutors in their role and SW students in practice are deliberated.
    • Population Health Screening after Environmental Pollution

      Stewart, Alex G.; orcid: 0000-0002-4931-5340; email: dragonsteeth@doctors.org.uk; Wilkinson, Ewan; orcid: 0000-0002-2167-8756; email: ewilkinson@chester.ac.uk (MDPI, 2020-11-24)
      Following environmental pollution exposure, calls to screen the population for disease or disease markers are often made. Population screening is a cross-sectional review of a population to find latent cases or biomarkers of disease that indicate the possibility of disease development; it differs from environmental screening or an epidemiological survey. Recognized standard approaches have been developed over 60 years to ensure quality and effectiveness in complex programs. We surveyed the literature for papers on health screening following environmental exposures and checked them for reference to accepted criteria such as those of Wilson and Jungner. We applied these criteria to three situations covering source/hazard (arsenic contaminated land), pathway/exposure (radiation release), and receptor/disease (lead poisoning). We identified 36 relevant papers. Although across the papers the whole range of criteria were addressed, no paper or program utilized recognized criteria. Issues and gaps identified included limited strategic approaches, lack of treatment, environmental prevention being seen as the screening outcome instead of treatment of identified individuals, and programs which did not fit the World Health Organization screening description. Robust discussion in the literature is needed to consider the organization and role of health screening following environmental exposures.
    • The cat is out of the bag – point-of-care testing (POCT) is here to stay

      Phin, Nick; Poutanen, Susan M (European Centre for Disease Control and Prevention (ECDC), 2020-11-05)
    • Problem Joints and Their Clinical and Humanistic Burden in Children and Adults with Moderate and Severe Hemophilia a: CHESS Paediatrics and CHESS II

      McLaughlin, Paul; Hermans, Cedric; Asghar, Sohaib; Burke, Tom; Nissen, Francis; Aizenas, Martynas; Meier, Oliver; Dhillon, Harpal; O'Hara, Jamie (American Society of Hematology, 2020-11-05)
      Introduction Severe hemophilia A (SHA) is characterized by spontaneous (non-trauma related) bleeding episodes into the joint space and muscle tissue, leading to progressive joint deterioration and chronic pain. Chronic joint damage is most often associated with severe hemophilia, however more recent research has illustrated that people with moderate hemophilia A (MHA) also experience hemophilic arthropathy and functional impairment. The need to measure joint health in children as well as adults, is underscored by findings from the Joint Outcome Continuation Study, which found that FVIII prophylaxis was insufficient to protect joints from damage, from childhood through adolescence in severe HA (Warren et al., 2020). The objective of this analysis is to gain a more patient-centric understanding of the clinical, economic and humanistic burden associated with 'Problem Joints', a measure of joint morbidity developed in consultation with an expert panel to overcome limitations with existing measures, in people with MHA and SHA. Methods A descriptive cohort analysis was conducted, utilizing retrospective, cross-sectional real-world data from the 'Cost of Haemophilia in Europe: a Socioeconomic Survey' (CHESS Paeds and CHESS II), studies of adult and pediatric persons with hemophilia. The analysis population is comprised of children (17 and below) with MHA or SHA in CHESS Paeds, and adults aged 20 and over with MHA or SHA in CHESS II. To account for the possibility that persons aged 18 or 19 in CHESS II may have participated in CHESS Paeds, these individuals were excluded from the analysis. Physician-reported clinical outcome data and patient/caregiver-reported quality of life were analyzed. A problem joint (PJ) is defined as having chronic joint pain and/or limited range of movement due to compromised joint integrity (i.e. chronic synovitis and/or hemophilic arthropathy). Analyses were stratified by number of PJs: none, 1 PJ, and 2+ PJs. We report retrospective data of the 12 months prior to study enrollment, on annualized bleeding rate (ABR), prevalence of target joints (TJ), as defined by the International Society on Thrombosis and Haemostasis, and EQ-5D-/5L/Y/Proxy score. Results are presented as mean (standard deviation) or N (%). Results Among 785 participants (N = 464 SHA; N = 321 MHA) in CHESS Paeds, mean age and BMI were 10.33 (4.63) and 22.50 (17.07), respectively. Of 493 participants (aged 20 and above) in CHESS II (N = 298 SHA; N = 195 MHA), the mean age and BMI were 38.61 (14.06) and 24.55 (2.92), respectively. Current inhibitor to FVIII replacement was more prevalent in children than in adults (10% vs. 5%). In CHESS II, approximately 40% of people with MHA and 49% with SHA had one or more PJs, respectively [1 PJ (23% vs. 28%); 2+ PJs (16% vs. 21%)]. In CHESS Paeds, approximately 14% of children with MHA and 18% with SHA had at least one PJ, respectively [1 PJ (9% vs. 14%); 2+ PJs (5% vs. 3%)]. TJs were less prevalent with MHA in comparison to SHA, in both adults (24% vs. 45%) and children (13% vs. 22%). Clinical burden was higher among both children and adults with PJs compared to those with no PJs. ABR correlates with the number of PJs, in those with MHA and SHA in CHESS II (Figure 1). Similarly, PJs were associated with higher ABR across MHA and SHA in CHESS Paeds (Figure 2). Hemophilia-related hospitalizations were higher in both adult and pediatric participants with PJs. In CHESS II, MHA with no PJs had fewer [0.73 (1.23)] hospitalizations compared to having those with 1 PJ [1.38 (1.11)] or 2+ PJs [1.28 (1.25)]. Similarly, children with MHA with 2+ PJs had 1.60 (1.92) hemophilia-related hospitalizations, compared to 1.38 (1.92) with 1 PJ and 0.71 (1.14) with no PJs. PJs were associated with impaired quality of life. In CHESS II, MHA and SHA EQ-5D-5L values in persons with no PJs were 0.81 (0.19) and 0.79 (0.18), respectively, compared to 0.65 (0.16) and 0.62 (0.23) with 1 PJ, and 0.65 (0.14) and 0.51 (0.33) in with 2+ PJs. A similar trend was observed in EQ-5D-Y and EQ-5D-proxy scores in CHESS Paeds. Conclusions Data from CHESS Paeds and CHESS II demonstrate an association between chronic joint damage, as measured by the 'problem joint' definition, and worsening clinical and quality of life outcomes, across both MHA and SHA. Further analyses will seek to expand upon the initial results presented here, to investigate the wider elements of burden associated with compromised long-term joint health. Disclosures McLaughlin: BioMarin: Consultancy; Novo Nordisk: Consultancy, Speakers Bureau; Sobi: Consultancy, Speakers Bureau; Roche/Chugai: Speakers Bureau; Takeda: Speakers Bureau. Hermans:Novo Nordisk: Consultancy, Speakers Bureau; Roche: Consultancy, Speakers Bureau; Sobi: Consultancy, Research Funding, Speakers Bureau; Biogen: Consultancy, Speakers Bureau; CAF-DCF: Consultancy, Speakers Bureau; CSL Behring: Consultancy, Speakers Bureau; Shire, a Takeda company: Consultancy, Research Funding, Speakers Bureau; Pfizer: Consultancy, Research Funding, Speakers Bureau; Bayer: Consultancy, Research Funding, Speakers Bureau; WFH: Other; EAHAD: Other; Octapharma: Consultancy, Speakers Bureau; Kedrion: Speakers Bureau; LFB: Consultancy, Speakers Bureau. Asghar:HCD Economics: Current Employment. Burke:HCD Economics: Current Employment; University of Chester: Current Employment; F. Hoffmann-La Roche Ltd: Consultancy. Nissen:GSK: Research Funding; Novartis: Research Funding; Actelion: Consultancy; F. Hoffmann-La Roche Ltd: Current Employment. Aizenas:F. Hoffmann-La Roche Ltd: Current Employment, Current equity holder in publicly-traded company. Meier:F. Hoffmann-La Roche Ltd: Current Employment, Current equity holder in publicly-traded company. Dhillon:HCD Economics: Current Employment; F. Hoffmann-La Roche Ltd: Other: All authors received editorial support for this abstract, furnished by Scott Battle, funded by F. Hoffmann-La Roche Ltd, Basel, Switzerland. . O'Hara:F. Hoffmann-La Roche Ltd: Consultancy; HCD Economics: Current Employment, Current equity holder in private company.
    • Examination and Validation of a Patient-Centric Joint Metric: "Problem Joint"; Empirical Evidence from the CHESS US Dataset

      Burke, Tom; Rodriguez Santana, Idaira; Chowdary, Pratima; Curtis, Randall; Khair, Kate; Laffan, Michael; McLaughlin, Paul; Noone, Declan; O'Mahony, Brian; Pasi, John; et al. (American Society of Hematology, 2020-11-05)
      Introduction Severe hemophilia (FVIII/FIX level <1%) is characterized by spontaneous hemarthrosis leading to progressive joint deterioration and chronic pain in the affected individual. Unless these recurrent hemarthroses can be prevented, e.g. with the use of prophylactic factor replacement therapy, these patients will develop chronic synovitis, pain, and eventually destruction of the joint. Current metrics such as 'Target joint' and other clinical measures of joint morbidity are prevalent and widely accepted. Measures focused solely on bleeding activity, such as the 'Target joint' metric, are arguably becoming less sensitive as current treatment strategies look to significantly reduce or eradicate joint bleeds, though they maintain clinically relevant and complementary to delivery of comprehensive hemophilia care. Key opinion leaders in the haemophilia field have debated the need for a more patient relevant measure of haemophilia-related joint morbidity. 'Problem Joint' (PJ), which is defined as having chronic joint pain and/or limited range of movement due to compromised joint integrity (chronic synovitis and/or haemophilic arthropathy), with or without persistent bleeding was derived through consensus. The objectives of this working group are to examine the usefulness and validity of the PJ metric. Initial research presented here was used to test the sensitivity of PJ as a patient relevant metric with respect to key outcomes for US haemophilia patients. Methods Data on PJs, as well as demographic, clinical and socio-economic variables was captured within the 'Cost of Haemophilia Across Europe: A Socioeconomic Survey' datasets (CHESS: I, II, Paediatric, and US studies). These data contain a total of 992 paediatric (age 1-17) and 2,437 adults (age 18+) with haemophilia from eight European countries and the US. Statistical analysis explored the association of PJ count and location with respect to two key outcomes: quality of life, as measured by an EQ-5D score, and overall work impairment, measured by the Work Productivity and Activity Impairment Questionnaire (WPAI). Those with current inhibitors were excluded from the analysis, and the US cohort comprised the focus of this initial research into the topic. Results The US cohort contained information on 345 people with haemophilia (PwH) and captured adults only, with a mean age of 35 years. Approximately, 43% of PwH had one or more PJs. Lower body PJs were more prevalent than upper body: 40% had one or more lower body PJs vs. 27% upper body. The majority of PJs were located in the ankles, knees and elbows. The relationship between EQ-5D and number of PJs showed a negative trend (see Figure 1): the average EQ-5D score was: 0.81 for those with zero PJs (N=197); 0.79 for those with one PJ (N=24); 0.70 for two PJs (N=29); 0.68 for three PJs (N=24) and 0.49 for those patients with four of more PJs (N=59). Similarly, an increase in number of PJs meant greater work productivity impairment versus no PJs recorded: 30.08% (N=102) vs. 19.51% (N=137), respectively. Discussion Results from the US cohort found that an increase in the number of PJs was associated with an increasing humanistic burden in PwH. The proposed Problem Joint definition takes a holistic viewpoint and provides a patient relevant perspective. Further work is planned to evaluate the appropriateness of the measure, and test the sensitivity in European and pediatric cohorts. Disclosures Burke: HCD Economics: Current Employment; University of Chester: Current Employment; F. Hoffmann-La Roche Ltd: Consultancy. Rodriguez Santana:HCD Economics: Current Employment. Chowdary:Pfizer: Membership on an entity's Board of Directors or advisory committees, Research Funding, Speakers Bureau; Sobi: Membership on an entity's Board of Directors or advisory committees, Research Funding, Speakers Bureau; Roche: Membership on an entity's Board of Directors or advisory committees, Speakers Bureau; Sanofi: Membership on an entity's Board of Directors or advisory committees; Shire (Baxalta): Membership on an entity's Board of Directors or advisory committees; Spark: Membership on an entity's Board of Directors or advisory committees; BioMarin: Honoraria; Novo Nordisk: Membership on an entity's Board of Directors or advisory committees, Research Funding, Speakers Bureau; CSL Behring: Membership on an entity's Board of Directors or advisory committees, Research Funding, Speakers Bureau; Chugai: Membership on an entity's Board of Directors or advisory committees, Speakers Bureau; Freeline: Membership on an entity's Board of Directors or advisory committees, Research Funding; Bayer: Membership on an entity's Board of Directors or advisory committees, Research Funding. Curtis:Bayer: Consultancy; Novo Nordisk: Consultancy; Patient Reported Outcomes, Burdens and Experiences: Consultancy; USC Hemophilia Utilization Group Study (HUGS): Consultancy. Khair:Haemnet: Membership on an entity's Board of Directors or advisory committees; Biomarin: Consultancy; HCD Economics: Consultancy; Novo Nordisk: Consultancy, Membership on an entity's Board of Directors or advisory committees; Medikhair: Membership on an entity's Board of Directors or advisory committees; Sobi: Consultancy, Honoraria, Research Funding, Speakers Bureau; CSL Behring: Honoraria, Research Funding; F. Hoffmann-La Roche Ltd: Honoraria, Research Funding; Takeda: Honoraria, Speakers Bureau; Bayer: Consultancy, Honoraria, Speakers Bureau. Laffan:Shire: Consultancy; LFB: Consultancy; Roche: Consultancy; Sobi: Consultancy; Pfizer: Consultancy; CSL: Consultancy; Pfizer: Speakers Bureau; Bayer: Speakers Bureau; Roche-Chugai: Speakers Bureau; Takeda: Speakers Bureau; Leo-Pharma: Speakers Bureau; Octapharma: Consultancy. McLaughlin:BioMarin: Consultancy; Novo Nordisk: Consultancy, Speakers Bureau; Sobi: Consultancy, Speakers Bureau; Roche/Chugai: Speakers Bureau; Takeda: Speakers Bureau. Noone:European Haemophilia Consortium: Membership on an entity's Board of Directors or advisory committees; Research Investigator PROBE: Research Funding; Healthcare Decision Consultants: Membership on an entity's Board of Directors or advisory committees. O'Mahony:Biomarin: Honoraria, Membership on an entity's Board of Directors or advisory committees; Freeline: Honoraria; UniQure: Honoraria. Pasi:BioMarin: Consultancy, Honoraria, Other: Grants, personal fees, and nonfinancial support; honoraria as member of scientific advisory boards and symposia; uniQure: Other: Grants and nonfinancial support , Research Funding; ApcinteX: Consultancy, Other: Personal fees ; Octapharma: Honoraria, Other: Personal fees and nonfinancial support; honoraria as member of scientific advisory boards and symposia , Speakers Bureau; Novo Nordisk: Honoraria, Other: Personal fees and nonfinancial support; honoraria as member of scientific advisory boards and symposia ; Catalyst Biosciences: Consultancy, Other: Personal fees and nonfinancial support; honoraria as member of scientific advisory boards and symposia; Biotest: Consultancy, Honoraria, Other: Personal fees and nonfinancial support; honoraria as member of scientific advisory boards and symposia; Alnylam (Sanofi): Other: Personal fees and nonfinancial support ; Takeda: Consultancy, Honoraria, Other: Personal fees; honoraria as member of scientific advisory boards and symposia ; Sanofi: Honoraria, Other: Personal fees and nonfinancial support; honoraria as member of scientific advisory boards and symposia, Research Funding; Sigilon: Research Funding; Tremeau: Research Funding; Sobi: Consultancy, Honoraria, Other; Roche: Honoraria, Other; Pfizer: Other. Skinner:Genentech: Consultancy, Honoraria; Spark Therapeutics: Other, Speakers Bureau; Pfizer: Other, Speakers Bureau; Takeda: Honoraria, Research Funding; uniQure: Research Funding; Biomarin: Consultancy, Research Funding; CSL Behring: Research Funding; Freeline Therapeutics: Research Funding; Novo Nordisk: Honoraria, Research Funding; Roche: Honoraria, Research Funding; Sanofi: Honoraria, Research Funding, Speakers Bureau; Sobi: Research Funding; Bayer: Consultancy, Research Funding. O'Hara:HCD Economics: Current Employment, Current equity holder in private company; F. Hoffmann-La Roche Ltd: Consultancy.